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Fibrosis of Extraocular Muscles, CFEOM5

OMIM ID:

autosomal recessive

Fibrosis of Extraocular Muscles, CFEOM5

Alternate Names

CFEOM5
congenital cranial dysinnervation disorder

Defective Genes

COL25A1

Clinical Characteristics

Ocular Features

This type of congenital fibrosis of extraocular muscles is sometimes called a congenital cranial dysinnervation disorder.  Ptosis is of congenital onset while the nature of the strabismus is variable but bilateral.  One sib with this disorder had Duane retraction syndrome.

Systemic Features

No systemic features have been reported.

Genetics

Inheritance

Homozygosity or compound heterozygosity of mutations in the COL25A1 gene is responsible for this autosomal recessive condition. 

Other nonsyndromal forms of congenital fibrosis of extraocular muscles include: CFEOM1 (135700), CFEOM2 (602078), CFEOM3C (609384), and CFEOM with synergistic divergence (609612).  See also Tukel CFEOM syndrome (609428).

Pedigree

Autosomal recessive

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent.  Carriers with only one mutation, such as the parents, do not have clinical disease.  Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Image
Sample pedigree of autosomal recessive inheritance

In order for autosomal recessive disorders to be expressed, offspring generally must inherit two mutations, one from each carrier parent. Carriers with only one mutation, such as the parents, do not have clinical disease. Note that carrier parents can expect that 1 in 4 children (25%) will inherit both mutations and have the disorder, 2 in 4 children (50%) will be carriers like their parents, while 1 in 4 children (25%) inherit neither mutation.

Treatment & Management

No specific treatment has been reported.  However, in selected patients the ocular deviation can be at least partially corrected with strabismus surgery.  Surgery for ptosis should also be considered.

Selected Resources

Publications

Displaying 1 - 1 of 1

Recessive Mutations in COL25A1 Are a Cause of Congenital Cranial Dysinnervation Disorder

PubMedID: 25500261